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CIC-DUX4 Sarcoma Involving the Skull Base: A Rare Presentation and Review of the Literature

Background  CIC-DUX4 sarcoma is a rare, aggressive tumor that is difficult to diagnose. Although it is closely related to Ewing's sarcoma, each is a distinct pathologic entity and both have been previously reported in the skin, lymph nodes, and viscera. We report the first description of CIC-DU...

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Autores principales: Hobday, Sara B., Mady, Leila J., Jacobson, Alec M., Rassekh, Christopher H.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Georg Thieme Verlag KG 2023
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10575739/
https://www.ncbi.nlm.nih.gov/pubmed/37842548
http://dx.doi.org/10.1055/a-2166-5688
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author Hobday, Sara B.
Mady, Leila J.
Jacobson, Alec M.
Rassekh, Christopher H.
author_facet Hobday, Sara B.
Mady, Leila J.
Jacobson, Alec M.
Rassekh, Christopher H.
author_sort Hobday, Sara B.
collection PubMed
description Background  CIC-DUX4 sarcoma is a rare, aggressive tumor that is difficult to diagnose. Although it is closely related to Ewing's sarcoma, each is a distinct pathologic entity and both have been previously reported in the skin, lymph nodes, and viscera. We report the first description of CIC-DUX4 involving the posterior cranial fossa and review the distinctive symptomatology, morphology, immunoprofile, and genetic signature that differentiate this rare tumor. Case Report  A 32-year-old man presented with an enlarging right lateral neck mass, progressive hoarseness, and orofacial pain. Biopsy revealed a high-grade undifferentiated malignant neoplasm. Imaging demonstrated an 8-cm mass in the right neck extending to the skull base and abutting the carotid sheath, in addition to pulmonary nodules and pelvic lymphadenopathy. Despite initial response to chemotherapy, he experienced disease progression and underwent surgical resection, radical neck dissection, and brachytherapy. Definitive pathologic diagnosis was achieved with next-generation sequencing. Within weeks of treatment, he developed symptoms reflecting progression of disease involving the neck, posterior cranial fossa, and lung. Adjuvant chemotherapy was planned, but the patient succumbed to his disease prior to initiation of further therapy. Conclusion  CIC-DUX4 sarcomas are uncommon and can progress rapidly. Diagnosis requires either fluorescence in situ hybridization or next-generation sequencing. Due to its rarity, there is no standard-of-care treatment for this tumor and further investigations are needed to understand disease behavior and develop targeted therapeutic modalities.
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spelling pubmed-105757392023-10-14 CIC-DUX4 Sarcoma Involving the Skull Base: A Rare Presentation and Review of the Literature Hobday, Sara B. Mady, Leila J. Jacobson, Alec M. Rassekh, Christopher H. J Neurol Surg Rep Background  CIC-DUX4 sarcoma is a rare, aggressive tumor that is difficult to diagnose. Although it is closely related to Ewing's sarcoma, each is a distinct pathologic entity and both have been previously reported in the skin, lymph nodes, and viscera. We report the first description of CIC-DUX4 involving the posterior cranial fossa and review the distinctive symptomatology, morphology, immunoprofile, and genetic signature that differentiate this rare tumor. Case Report  A 32-year-old man presented with an enlarging right lateral neck mass, progressive hoarseness, and orofacial pain. Biopsy revealed a high-grade undifferentiated malignant neoplasm. Imaging demonstrated an 8-cm mass in the right neck extending to the skull base and abutting the carotid sheath, in addition to pulmonary nodules and pelvic lymphadenopathy. Despite initial response to chemotherapy, he experienced disease progression and underwent surgical resection, radical neck dissection, and brachytherapy. Definitive pathologic diagnosis was achieved with next-generation sequencing. Within weeks of treatment, he developed symptoms reflecting progression of disease involving the neck, posterior cranial fossa, and lung. Adjuvant chemotherapy was planned, but the patient succumbed to his disease prior to initiation of further therapy. Conclusion  CIC-DUX4 sarcomas are uncommon and can progress rapidly. Diagnosis requires either fluorescence in situ hybridization or next-generation sequencing. Due to its rarity, there is no standard-of-care treatment for this tumor and further investigations are needed to understand disease behavior and develop targeted therapeutic modalities. Georg Thieme Verlag KG 2023-10-13 /pmc/articles/PMC10575739/ /pubmed/37842548 http://dx.doi.org/10.1055/a-2166-5688 Text en The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. ( https://creativecommons.org/licenses/by-nc-nd/4.0/ ) https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives License, which permits unrestricted reproduction and distribution, for non-commercial purposes only; and use and reproduction, but not distribution, of adapted material for non-commercial purposes only, provided the original work is properly cited.
spellingShingle Hobday, Sara B.
Mady, Leila J.
Jacobson, Alec M.
Rassekh, Christopher H.
CIC-DUX4 Sarcoma Involving the Skull Base: A Rare Presentation and Review of the Literature
title CIC-DUX4 Sarcoma Involving the Skull Base: A Rare Presentation and Review of the Literature
title_full CIC-DUX4 Sarcoma Involving the Skull Base: A Rare Presentation and Review of the Literature
title_fullStr CIC-DUX4 Sarcoma Involving the Skull Base: A Rare Presentation and Review of the Literature
title_full_unstemmed CIC-DUX4 Sarcoma Involving the Skull Base: A Rare Presentation and Review of the Literature
title_short CIC-DUX4 Sarcoma Involving the Skull Base: A Rare Presentation and Review of the Literature
title_sort cic-dux4 sarcoma involving the skull base: a rare presentation and review of the literature
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10575739/
https://www.ncbi.nlm.nih.gov/pubmed/37842548
http://dx.doi.org/10.1055/a-2166-5688
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